Gingival leiomyomatous hamartoma of the maxilla: a rare entity

Abstract

Hamartoma is a tumour-like malformation appearing as a focal overgrowth of normal cells. Leiomyomatous hamartomas (LHs) are rare in the oral cavity and commonly seen in the Japanese and less than 40 cases have been reported in the Japanese and English literature. The clinical differential diagnoses are irritational (traumatic) fibroma and congenital epulis. It has to be differentiated histopathologically from its neoplastic counterparts and mesenchymomas. Hence, we report such a case of LHs, which presented as a sessile gingival growth occurring in the midline in a 15-year-old girl. The final diagnosis was based on the histopathological appearance which was confirmed by immunohistochemical staining of various markers. A review of the literature of previous cases was also carried out.

Leiomyomatous Hamartomas of the Oral Cavity: Clinicopathological and Immunohistochemical Features of 4 Cases and Literature Review

“…8,9 In the maxillary cases diagnosed after infancy, teeth displacement was present. 10,11 The midline tongue (mainly tongue base or foramen cecum region) and the upper midline gingiva/alveolar ridge are considered fusion regions of embryologic tissues, which could explain the predilection of OLH for these sites. There are also reports in the Japanese literature of palatal depression corresponding to the OLH mass in the tongue, suggesting that the mass was present in the period of embryonal development when the secondary palate was formed.…”

“…In older patients, fibroma, pyogenic granuloma, peripheral ossifying fibroma, and peripheral giant cell granuloma should be considered in the differential diagnosis. 2,11,16 Some authors have mentioned possible difficulties in the histopathological differentiation between lingual hamartomas-with 2 or more mesenchymal components-and benign mesenchymomas; although true distinction in some cases may represent only a terminology issue, differently from benign mesenchymomas, most OLH are characterized by the predominance of one tissue type, lack of infiltration or invasion to surrounding tissues, location restricted to dorsum of tongue and anterior maxilla, and congenital nature or diagnosis in the first years of life. The term OLH is well accepted and used for this entity in the vast majority of the studies (Table 1).…”

“…The term OLH is well accepted and used for this entity in the vast majority of the studies (Table 1). 1,9,11 The treatment of OLH is conservative surgical excision. The prognosis is good and no recurrences are expected, as shown by the current cases.…”

“…In older patients, fibroma, pyogenic granuloma, peripheral ossifying fibroma, and peripheral giant cell granuloma should be considered in the differential diagnosis. 2,11,16…”

“…The term OLH is well accepted and used for this entity in the vast majority of the studies (Table 1). 1,9,11…”